Bollerslev J, Kvetny J
Department of Internal Medicine, Svendborg Hospital.
Scand J Clin Lab Invest. 1988 Dec;48(8):795-9. doi: 10.3109/00365518809088762.
Seven patients with autosomal dominant osteopetrosis from three different families were investigated. They all had the roentgenological type I disease, characterized by universal, symmetrical osteosclerosis and enlarged thickness of the calvarium vault. Serum concentrations of thyroid hormones and the specific nuclear binding of triiodothyronine (T3) in mononuclear blood cells were studied. All patients appeared clinically euthyroid. The serum level of T3 was significantly elevated (serum T3 = 1.89 nmol/l) compared with normal age and sex-matched controls (serum T3 = 1.44 nmol/l, p less than 0.05). The specific maximal nuclear binding capacity of T3 was significantly decreased (MBC = 0.51 fmol T3/mg DNA) in these patients compared with controls (MBC = 1.8 fmol/mg DNA, p less than 0.05) whereas no difference in the equilibrium association constant (Ka) was observed. The decreased specific nuclear binding of T3 and the slightly elevated serum level of T3 might indicate a modest peripheral resistance to T3 in patients with autosomal dominant osteopetrosis type I.
对来自三个不同家族的7例常染色体显性遗传性骨硬化症患者进行了研究。他们均患有I型放射学疾病,其特征为全身性、对称性骨硬化以及颅盖厚度增加。研究了甲状腺激素的血清浓度以及单核血细胞中三碘甲状腺原氨酸(T3)的特异性核结合情况。所有患者临床均表现为甲状腺功能正常。与年龄和性别匹配的正常对照组相比,患者的血清T3水平显著升高(血清T3 = 1.89 nmol/l)(正常对照组血清T3 = 1.44 nmol/l,p < 0.05)。与对照组相比,这些患者的T3特异性最大核结合能力显著降低(MBC = 0.51 fmol T3/mg DNA)(对照组MBC = 1.8 fmol/mg DNA,p < 0.05),而平衡结合常数(Ka)未观察到差异。I型常染色体显性遗传性骨硬化症患者T3特异性核结合降低以及血清T3水平轻度升高可能提示存在一定程度的外周T3抵抗。